Yuan CY, Zhang ZR, Guo MF, Zhang N. Recurrent multisystem Langerhans cell histiocytosis involving the female genitalia: A case report. World J Clin Cases 2024; 12(28): 6222-6229 [PMID: 39371561 DOI: 10.12998/wjcc.v12.i28.6222]
Corresponding Author of This Article
Na Zhang, MM, Doctor, Department of Gynecologic Oncology, Chongqing Key Laboratory of Translational Research for Cancer Metastasis and Individualized Treatment, Chongqing University Cancer Hospital, Chongqing Cancer Institute, Chongqing Cancer Hospital, No. 181 Hanyu Road, Shapingba District, Chongqing 400030, China. zhang_zhang07@163.com
Research Domain of This Article
Obstetrics & Gynecology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
World J Clin Cases. Oct 6, 2024; 12(28): 6222-6229 Published online Oct 6, 2024. doi: 10.12998/wjcc.v12.i28.6222
Recurrent multisystem Langerhans cell histiocytosis involving the female genitalia: A case report
Chun-Yan Yuan, Zhi-Rong Zhang, Ming-Fang Guo, Na Zhang
Chun-Yan Yuan, Zhi-Rong Zhang, Department of Gynaecology, Fengdu People's Hospital, Chongqing 408200, China
Ming-Fang Guo, Na Zhang, Department of Gynecologic Oncology, Chongqing Key Laboratory of Translational Research for Cancer Metastasis and Individualized Treatment, Chongqing University Cancer Hospital, Chongqing Cancer Institute, Chongqing Cancer Hospital, Chongqing 400030, China
Co-corresponding authors: Ming-Fang Guo and Na Zhang.
Author contributions: Yuan CY and Zhang ZR were involved in data collection; Zhang N and Guo MF were involved in the supervision of the study and conceptualized and designed the research; Zhang N and Guo MF performed data analysis; Yuan CY, Zhang N and Guo MF wrote the paper; Guo MF applied for and obtained the funds for this research project. Guo MF conceptualized, designed, and supervised the whole process of the project. Zhang N was instrumental and responsible for data re-analysis and re-interpretation, figure plotting, comprehensive literature search, preparation and submission of the current version of the manuscript. Both Zhang N and Guo MF have played important and indispensable roles in the experimental design, data interpretation and manuscript preparation as the co-corresponding authors. This collaboration between Zhang N and Guo MF is crucial for the publication of this manuscript and other manuscripts still in preparation. And all authors have reviewed the final version to be published and agreed to be accountable for all aspects of the work.
Supported bythe Chongqing Science and Health Committee Joint Medical Scientific Research General Project, No. 2021MSXM236; and the Health Development Promotion Project, No. BJHA-CRP-086.
Informed consent statement: The study participant provided informed written consent prior to study registration.
Conflict-of-interest statement: All authors declare that they have no conflict of interest to disclose.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Na Zhang, MM, Doctor, Department of Gynecologic Oncology, Chongqing Key Laboratory of Translational Research for Cancer Metastasis and Individualized Treatment, Chongqing University Cancer Hospital, Chongqing Cancer Institute, Chongqing Cancer Hospital, No. 181 Hanyu Road, Shapingba District, Chongqing 400030, China. zhang_zhang07@163.com
Received: March 23, 2024 Revised: May 20, 2024 Accepted: June 20, 2024 Published online: October 6, 2024 Processing time: 142 Days and 15.6 Hours
Abstract
BACKGROUND
Langerhans cell histiocytosis (LCH) is a histiocytic proliferative disease caused by clonal proliferation of Langerhans cells, which is currently defined as an inflammatory myeloid tumor. It is rare in adults, with an incidence of 1–2 per million, and is highly heterogeneous in clinical presentation, with unpredictable disease progression and outcome.
CASE SUMMARY
A 52-year-old postmenopausal female patient presented to the gynecology department in July 2023 with bilateral vulvar masses. She was diagnosed with recurrent multisystem LCH. The patient had previously been diagnosed with a single-system and single-focal LCH in October 2021 due to a right maxillofacial mass, which resolved after surgical treatment. A chemotherapy regimen was developed after multidisciplinary consultation. Six cycles of chemotherapy resulted in partial remission, and maintenance chemotherapy is currently being administered.
CONCLUSION
Recurrent LCH involving the bilateral vulva has been poorly reported. Comprehensive imaging and pathological evaluation is important for diagnosis. The model of joint multidisciplinary specialist diagnosis and treatment is worthy of clinical application.
Core Tip: The pathogenesis of Langerhans cell histiocytosis (LCH) is unclear and it is currently classified as a neoplastic disease. Recurrent LCH involving the female genitalia is rare. Pathological diagnosis is considered the gold standard. For patients with extensive lesions or relapses, we recommend a hematology-based multidisciplinary diagnostic and treatment model. New combination chemotherapy regimens and maintenance chemotherapy may result in better clinical outcomes.
